Sleep disorders in myotonic dystrophy type 2: A controlled polysomnographic study and self-reported questionnaires

A. Romigi, M. Albanese, F. Placidi, F. Izzi, C. Liguori, M. G. Marciani, N. B. Mercuri, C. Terracciano, G. Vitrani, A. Petrucci, B. Di Gioia, R. Massa

Research output: Contribution to journalArticle

Abstract

Background and purpose: There is a paucity of data available regarding the occurrence of sleep disorders in myotonic dystrophy type 2 (DM2). In this study the sleep-wake cycle and daytime sleepiness were investigated in DM2 patients and compared with results from healthy subjects and myotonic dystrophy type 1 (DM1) patients. Methods: Twelve DM2 outpatients, 12 age- and sex-matched healthy controls and 18 DM1 patients were recruited. Subjective quality of sleep was assessed by means of the Pittsburgh Sleep Quality Index (PSQI). Both the Epworth Sleepiness Scale and the Daytime Sleepiness Scale were performed in order to evaluate excessive daytime sleepiness (EDS). All participants underwent polysomnographic monitoring over 48 h as well as the Multiple Sleep Latency Test. Results: Sleep efficiency was <90% in 12/12 DM2 patients, and significantly reduced when compared with controls or with DM1. Decreased sleep efficiency was associated with sleep-disordered breathing in seven out of 12 DM2 patients and/or periodic limbs movements of sleep (PLMS) in three out of eight patients. Six DM2 patients showed REM sleep without atonia, whereas none of the controls or DM1 patients showed REM sleep dysregulation. The global PSQI score was higher in DM2 patients than in controls and DM1 patients. Conclusions: Sleep quality in DM2 patients is poorer than in DM1 patients and controls. Sleep apnea is the most common sleep disorder in DM2 patients. Obstructive sleep apnea and sleep fragmentation may represent the main cause of EDS, whereas PLMS is a frequent finding in DM1.

Original languageEnglish
Pages (from-to)929-934
Number of pages6
JournalEuropean Journal of Neurology
Volume21
Issue number6
DOIs
Publication statusPublished - 2014

Fingerprint

Myotonic Dystrophy
Sleep
REM Sleep
Sleep Apnea Syndromes
Surveys and Questionnaires
Sleep Wake Disorders
Extremities
Sleep Deprivation
Obstructive Sleep Apnea
Healthy Volunteers

Keywords

  • Excessive daytime somnolence
  • Multiple sleep latency test
  • Myotonic dystrophy
  • REM sleep

ASJC Scopus subject areas

  • Clinical Neurology
  • Neurology
  • Medicine(all)

Cite this

Sleep disorders in myotonic dystrophy type 2 : A controlled polysomnographic study and self-reported questionnaires. / Romigi, A.; Albanese, M.; Placidi, F.; Izzi, F.; Liguori, C.; Marciani, M. G.; Mercuri, N. B.; Terracciano, C.; Vitrani, G.; Petrucci, A.; Di Gioia, B.; Massa, R.

In: European Journal of Neurology, Vol. 21, No. 6, 2014, p. 929-934.

Research output: Contribution to journalArticle

Romigi, A, Albanese, M, Placidi, F, Izzi, F, Liguori, C, Marciani, MG, Mercuri, NB, Terracciano, C, Vitrani, G, Petrucci, A, Di Gioia, B & Massa, R 2014, 'Sleep disorders in myotonic dystrophy type 2: A controlled polysomnographic study and self-reported questionnaires', European Journal of Neurology, vol. 21, no. 6, pp. 929-934. https://doi.org/10.1111/ene.12226
Romigi, A. ; Albanese, M. ; Placidi, F. ; Izzi, F. ; Liguori, C. ; Marciani, M. G. ; Mercuri, N. B. ; Terracciano, C. ; Vitrani, G. ; Petrucci, A. ; Di Gioia, B. ; Massa, R. / Sleep disorders in myotonic dystrophy type 2 : A controlled polysomnographic study and self-reported questionnaires. In: European Journal of Neurology. 2014 ; Vol. 21, No. 6. pp. 929-934.
@article{0b30402a97904377b0757519b8217ba3,
title = "Sleep disorders in myotonic dystrophy type 2: A controlled polysomnographic study and self-reported questionnaires",
abstract = "Background and purpose: There is a paucity of data available regarding the occurrence of sleep disorders in myotonic dystrophy type 2 (DM2). In this study the sleep-wake cycle and daytime sleepiness were investigated in DM2 patients and compared with results from healthy subjects and myotonic dystrophy type 1 (DM1) patients. Methods: Twelve DM2 outpatients, 12 age- and sex-matched healthy controls and 18 DM1 patients were recruited. Subjective quality of sleep was assessed by means of the Pittsburgh Sleep Quality Index (PSQI). Both the Epworth Sleepiness Scale and the Daytime Sleepiness Scale were performed in order to evaluate excessive daytime sleepiness (EDS). All participants underwent polysomnographic monitoring over 48 h as well as the Multiple Sleep Latency Test. Results: Sleep efficiency was <90{\%} in 12/12 DM2 patients, and significantly reduced when compared with controls or with DM1. Decreased sleep efficiency was associated with sleep-disordered breathing in seven out of 12 DM2 patients and/or periodic limbs movements of sleep (PLMS) in three out of eight patients. Six DM2 patients showed REM sleep without atonia, whereas none of the controls or DM1 patients showed REM sleep dysregulation. The global PSQI score was higher in DM2 patients than in controls and DM1 patients. Conclusions: Sleep quality in DM2 patients is poorer than in DM1 patients and controls. Sleep apnea is the most common sleep disorder in DM2 patients. Obstructive sleep apnea and sleep fragmentation may represent the main cause of EDS, whereas PLMS is a frequent finding in DM1.",
keywords = "Excessive daytime somnolence, Multiple sleep latency test, Myotonic dystrophy, REM sleep",
author = "A. Romigi and M. Albanese and F. Placidi and F. Izzi and C. Liguori and Marciani, {M. G.} and Mercuri, {N. B.} and C. Terracciano and G. Vitrani and A. Petrucci and {Di Gioia}, B. and R. Massa",
year = "2014",
doi = "10.1111/ene.12226",
language = "English",
volume = "21",
pages = "929--934",
journal = "European Journal of Neurology",
issn = "1351-5101",
publisher = "Wiley-Blackwell Publishing Ltd",
number = "6",

}

TY - JOUR

T1 - Sleep disorders in myotonic dystrophy type 2

T2 - A controlled polysomnographic study and self-reported questionnaires

AU - Romigi, A.

AU - Albanese, M.

AU - Placidi, F.

AU - Izzi, F.

AU - Liguori, C.

AU - Marciani, M. G.

AU - Mercuri, N. B.

AU - Terracciano, C.

AU - Vitrani, G.

AU - Petrucci, A.

AU - Di Gioia, B.

AU - Massa, R.

PY - 2014

Y1 - 2014

N2 - Background and purpose: There is a paucity of data available regarding the occurrence of sleep disorders in myotonic dystrophy type 2 (DM2). In this study the sleep-wake cycle and daytime sleepiness were investigated in DM2 patients and compared with results from healthy subjects and myotonic dystrophy type 1 (DM1) patients. Methods: Twelve DM2 outpatients, 12 age- and sex-matched healthy controls and 18 DM1 patients were recruited. Subjective quality of sleep was assessed by means of the Pittsburgh Sleep Quality Index (PSQI). Both the Epworth Sleepiness Scale and the Daytime Sleepiness Scale were performed in order to evaluate excessive daytime sleepiness (EDS). All participants underwent polysomnographic monitoring over 48 h as well as the Multiple Sleep Latency Test. Results: Sleep efficiency was <90% in 12/12 DM2 patients, and significantly reduced when compared with controls or with DM1. Decreased sleep efficiency was associated with sleep-disordered breathing in seven out of 12 DM2 patients and/or periodic limbs movements of sleep (PLMS) in three out of eight patients. Six DM2 patients showed REM sleep without atonia, whereas none of the controls or DM1 patients showed REM sleep dysregulation. The global PSQI score was higher in DM2 patients than in controls and DM1 patients. Conclusions: Sleep quality in DM2 patients is poorer than in DM1 patients and controls. Sleep apnea is the most common sleep disorder in DM2 patients. Obstructive sleep apnea and sleep fragmentation may represent the main cause of EDS, whereas PLMS is a frequent finding in DM1.

AB - Background and purpose: There is a paucity of data available regarding the occurrence of sleep disorders in myotonic dystrophy type 2 (DM2). In this study the sleep-wake cycle and daytime sleepiness were investigated in DM2 patients and compared with results from healthy subjects and myotonic dystrophy type 1 (DM1) patients. Methods: Twelve DM2 outpatients, 12 age- and sex-matched healthy controls and 18 DM1 patients were recruited. Subjective quality of sleep was assessed by means of the Pittsburgh Sleep Quality Index (PSQI). Both the Epworth Sleepiness Scale and the Daytime Sleepiness Scale were performed in order to evaluate excessive daytime sleepiness (EDS). All participants underwent polysomnographic monitoring over 48 h as well as the Multiple Sleep Latency Test. Results: Sleep efficiency was <90% in 12/12 DM2 patients, and significantly reduced when compared with controls or with DM1. Decreased sleep efficiency was associated with sleep-disordered breathing in seven out of 12 DM2 patients and/or periodic limbs movements of sleep (PLMS) in three out of eight patients. Six DM2 patients showed REM sleep without atonia, whereas none of the controls or DM1 patients showed REM sleep dysregulation. The global PSQI score was higher in DM2 patients than in controls and DM1 patients. Conclusions: Sleep quality in DM2 patients is poorer than in DM1 patients and controls. Sleep apnea is the most common sleep disorder in DM2 patients. Obstructive sleep apnea and sleep fragmentation may represent the main cause of EDS, whereas PLMS is a frequent finding in DM1.

KW - Excessive daytime somnolence

KW - Multiple sleep latency test

KW - Myotonic dystrophy

KW - REM sleep

UR - http://www.scopus.com/inward/record.url?scp=84899910711&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=84899910711&partnerID=8YFLogxK

U2 - 10.1111/ene.12226

DO - 10.1111/ene.12226

M3 - Article

C2 - 23837695

AN - SCOPUS:84899910711

VL - 21

SP - 929

EP - 934

JO - European Journal of Neurology

JF - European Journal of Neurology

SN - 1351-5101

IS - 6

ER -